INT J LANG COMMUN DISORD, JANUARY–FEBRUARY VOL. 50, NO. 1, 119–128
2015,
Research Report Verbal competence in narrative retelling in 5-year-olds with unilateral cleft lip and palate Kristina Klint¨o†‡, Eva-Kristina Salameh‡ and Anette Lohmander†§ †Division of Speech and Language Pathology, Karolinska Institutet, Stockholm, Sweden ‡Department of Otorhinolaryngology, Sk˚ane University Hospital, Malm¨o, Sweden §Department of Speech Pathology, Karolinska University Hospital, Stockholm, Sweden
(Received January 2014; accepted July 2014) Abstract Background: Research regarding expressive language performance in children born with cleft palate is sparse. The relationship between articulation/phonology and expressive language skills also needs to be further explored. Aims: To investigate verbal competence in narrative retelling in 5-year-old children born with unilateral cleft lip and palate (UCLP) and its possible relationship with articulation/phonology at 3 and 5 years of age. Methods & Procedures: A total of 49 children, 29 with UCLP treated according to three different procedures for primary palatal surgery and a comparison group of 20 children (COMP), were included. Longitudinally recorded audio files were used for analysis. At ages 3 and 5, the children were presented with a single-word test of word naming and at age 5 also the Bus Story Test (BST). The BST was assessed according to a test manual. The single-word test was phonetically transcribed and the percentage of consonants correct adjusted for age (PCC-A) was calculated. Differences regarding the BST results within the UCLP group were analysed. The results were compared with the results of the COMP group, and also with norm values. In addition, the relationship between the results of the BST and the PCC-A scores at ages 3 and 5 years was analysed. Outcomes & Results: No significant group differences or correlations were found. However, 65.5% of the children in the UCLP group had an information score below 1 standard deviation from the norm value compared with 30% in the COMP group. Conclusions: A larger proportion of children in the UCLP group than in the COMP group displayed problems with retelling but the differences between the two groups were not significant. There was no association between the BST results in the children with UCLP and previous or present articulatory/phonological competence. Since group size was small in both groups, the findings need to be verified in a larger study. Keywords: cleft palate, expressive language, narrative, articulation, phonology.
What this paper adds? What is already known on this subject? About half of the children born with cleft palate have difficulties with speech and phonology at about 3 years of age. Earlier studies point towards a relationship between early limitations of speech/phonology and general linguistic constraints in children born with cleft palate. What this study adds? This study investigated verbal competence in narrative retelling at 5 years of age in children with unilateral cleft lip and palate (UCLP) relative to peers without cleft palate and its relationship to articulation/phonology. A larger proportion of children with UCLP than without displayed problems in the retelling task, although the difference
Address correspondence to: Kristina Klint¨o, Karolinska Institutet, Department of Clinical Science, Intervention, and Technique, CLINTEC, Division of Speech and Language Pathology, B69, Karolinska University Hospital, Huddinge, S-141 86 Stockholm, Sweden; e-mail: [email protected] International Journal of Language & Communication Disorders C 2014 Royal College of Speech and Language Therapists ISSN 1368-2822 print/ISSN 1460-6984 online DOI: 10.1111/1460-6984.12127
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was not statistically significant. The performance on the retelling task was not related to previous or present articulatory/phonological competence.
Introduction Language development in children born with cleft palate has been sparsely explored. Even though the cleft is repaired at an early age, some children have persisting problems with speech and phonology the first years (e.g., Chapman 1993, Klint¨o et al. 2014a, b). Moreover, in studies on toddlers with cleft palate, the results have indicated limited expressive language ability compared with toddlers without cleft palate (e.g., Scherer and D’Antonio 1995, Jocelyn et al. 1996, Broen et al. 1998). Early limitations in speech and phonology in toddlers with cleft palate may be related to general linguistic constraints (e.g., Chapman 2004, Frederickson et al. 2006). Problems with speech and phonology may lead to reduced intelligibility in speech, which could have a negative impact on language development. The genesis of language difficulties in children with cleft palate thus needs to be clarified, to ensure that the affected children get the adequate support early in life. It is therefore useful to investigate expressive language in children with cleft palate further, as well as to consider its possible relationship to articulatory and phonological competence. The number of studies on expressive language in children with cleft palate compared with peers without cleft palate at pre-school or early school ages is limited and these studies show varying results (Konst et al. 2003, Collett et al. 2010, Young et al. 2010, Chapman 2011). For example, Collett et al. (2010) and Chapman (2011) found no significant differences between 5- and 6-year-olds with and without cleft palate regarding expressive vocabulary and grammatical skills. In a study of 12 Dutch-speaking 6-year-olds with UCLP, vocabulary and syntactic skills were within the normal range (Konst et al. 2003). In contrast, Young et al. (2010) demonstrated difficulties in the expressive use of grammar and vocabulary (below the 20th percentile on a standardized screening tool) in about one-third of 43 Chinese Singaporean children with cleft palate between 3;9 and 6;8 years of age. Significant differences in lexical ability and mean length of utterance (MLU) have been found at younger ages, i.e. between toddlers (16–30 months of age) with and without cleft palate (Scherer and D’Antonio 1995). Jocelyn et al. (1996) compared 16 children with clefts and 16 children without clefts who were matched for sex, ethnicity, socioeconomic status and birth order. The children with clefts scored significantly lower on expressive language at 12 and 24 months of age as well as
on formal tests of cognition and comprehension. Broen et al. (1998) found that children with cleft palate scored significantly lower on cognitive and linguistic measures than children without clefts at 24 months of age, although within the normal range. Percentage of consonants correct (PCC) is a measure of articulatory/phonological errors (Shriberg 1993). In several studies, children with cleft palate as a group displayed significantly lower PCC scores than children without cleft palate at both 5 (Lohmander and Persson 2008) and 3 years of age (Lohmander and Persson 2008, Willadsen 2012, Klint¨o et al. 2014a). However, other studies have accounted for individual differences that may explain these scores. For example, Morris and Ozanne (2003) identified a subgroup among children with cleft palate who had phonological language impairment. The degree of phonological problems seems to some extent to be influenced by surgical treatment. In two studies where the cleft in the soft palate was closed around 4–5 months of age, children with unoperated cleft in the hard palate at 3 years of age displayed more restricted phonology than children with a closed cleft in the hard palate at the same age (Willadsen 2012, Klint¨o et al. 2014b). Results from Chapman (1993) indicate that differences in phonological ability decrease with age between children with cleft palate as a group and children without cleft palate. In 5-year-olds with cleft palate, the total instances of phonological processes were similar to peers born without clefts. However, only 10 children at age 5 years were included in the study, and the results need to be confirmed through more studies. Frederickson et al. (2006) studied the relationship between speech production skills and conversational style in children with cleft palate between 33 and 44 months of age. Among them, 35% were characterized as either low in assertiveness or low in responsiveness. A significant positive correlation was seen between speech production skills and conversational assertiveness. The authors suggested several explanations for a passive communication style. These included the effects of true pragmatic deficit, poor intelligibility due to poor speech leading to unwillingness to speak, and shy personality. Children with a passive communication style may have fewer opportunities to practise their language, e.g. in narratives. Narrative production has been reported to be an ecologically valid task when assessing language skills in children (Botting 2002). Narrative ability may predict persistent language impairment (Bishop and Edmundson
Verbal competence in 5-year-olds with cleft palate 1987) and literacy performance (Stothard et al. 1998). To reproduce a narrative, several underlying abilities are needed, such as understanding of the task and the relationship between the input text and the topic, remembrance of the text, and also processing of the input text at the required speed (Leinonen et al. 2000). Pragmatic skills are also essential (Botting 2002). Retelling generates longer and more complete stories and more complex story grammar than freely produced narratives (Merrit and Liles 1989). The Bus Story Test (BST) is a standardized retelling test for assessment of verbal competence, where children are asked to retell a story with the aid of 12 pictures. The test may also indicate problems with phonology, semantic/lexical ability, grammar, sequencing, and language comprehension (Renfrew 1997). The BST has been translated into Swedish based on normative data from 100 Swedish-speaking children aged between 3;9 and 6;8 (Svensson and Tuominen-Eriksson 2002). In an unpublished study of 60 Swedish-speaking children at 7 years of age who were born with cleft palate, significant problems were displayed when reproducing information compared with the norm values of children born without cleft palate (Abrahamsson 2005). However, no results on verbal competence when retelling in children with cleft palate have been published to our knowledge. Method and technique for surgery is an important variable that could influence development of speech and phonology (Willadsen 2012, Klint¨o et al. 2014b). Different surgical methods are practiced. Some researchers argue that facial growth benefits from delayed palatal closure (Friede 2007), while speech development benefits from palatal closure as early as possible (PetersonFalzone 1996). Currently in Sweden, four cleft palate centres perform surgery in two stages, with soft palate closure at about 6 months of age and hard palate closure at about two years of age, in order to promote facial growth. The two other centres perform surgery in one stage at about 12 or 15 months of age. For speech and language development in children born with non-syndromic cleft palate, the single most important factor in addition to the cleft is hearing. It is well recognized that the incidence of otitis media with effusion (OME) and related mild to moderate hearing loss is high among children born with cleft palate (e.g., Flynn et al. 2009). Early hearing status has been correlated both with comprehension scores and expressive language at 24 months of age (Jocelyn et al. 1996) and with number of consonant types at 12 months (Lohmander et al. 2011). Broen et al. (1998) observed differences in cognitive and receptive language skills by 24 months in children with and without cleft palate, which, however, disappeared when hearing status and velopharyngeal function at 12 months were included as covariates. At the same time, no clear correlation between hearing status and language skill has been found in older children with cleft palate.
121 In a study by Young et al. (2010), a positive history of OME was not found to influence expressive language in 5- and 6-year-olds with cleft palate. However, only four of the 43 children had a hearing loss on the day of assessment, and the influence of hearing was not analysed. Thus, adequate knowledge regarding expressive language ability in children with cleft palate is lacking, as is understanding of the influence of hearing status. Expressive language difficulties at pre-school and early school age could have an impact on communication and learning during the school years, and need to be further explored. Aims The aim of the present study was therefore to investigate expressive language skills in terms of verbal competence in narrative retelling in children with UCLP at age 5 years and to compare these skills with present and previous articulatory/phonological ability. The specific research questions were:
r How do 5-year-olds with UCLP perform on the information score, MLU, and the number of subordinate clauses when retelling, compared with children without cleft palate? r Are there any differences among children with UCLP related to gender or different methods for primary palatal surgery, regarding the information score, MLU, and the number of subordinate clauses when retelling at the age of 5 years? In addition, we assessed the relationship between the information score, MLU, and the number of subordinate clauses when retelling at 5 years of age and the PCC adjusted for age (PCC-A) at the ages of 5 and 3 years in the children born with UCLP. Methods This is a prospective comparative study of 5-year-old children with UCLP and a comparison group, and a retrospective comparison at 3 years of age. Participants The participants included a consecutively selected group of 20 children born with UCLP between 1997 and 2003 from the western region of Sweden, a consecutively selected group of 10 children born with UCLP between 2005 and 2008 from the southern region of Sweden, and a comparison (COMP) group of 20 children born without cleft lip and palate in 2000. The children with UCLP from the western region of Sweden were participants in the Scandcleft project (Semb 2001). The children in the COMP group were recruited from child health centres
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49 children
11 two-stage closure (hp-closure ≈ 12 months)
29 UCLP (12 girls+17 boys)
20 COMP (11 girls+9 boys)
9 two-stage closure (hp-closure ≈ 36 months)
9 one-stage closure (≈ 13 months)
Figure 1. Number of children in the different groups. UCLP, children with unilateral cleft lip and palate; COMP, children without cleft lip and palate; hp-closure, hard palate closure.
in Gothenburg. According to a parental questionnaire, the COMP group was considered to be typically developing, including in terms of language development. No formal cognitive test was used. All children were monolingual Swedish-speaking and without any known additional malformations or syndromes. In the group with UCLP from the southern region, the recording of one child at age 5 years was missing. This resulted in 29 children (12 girls and 17 boys) in the UCLP group and 20 children (11 girls and nine boys) in the COMP group (figure 1). The 20 children in the UCLP group from the western region had been treated with early soft palate repair together with lip closure at a mean age of 4.6 months (range = 3.4–6.4 months). Eleven of these children had then been treated with hard palate closure at a mean age of 12.3 months (range = 11.6–13.7 months), and the remaining nine with hard palate closure at a mean age of 36.2 months (range = 35.6–37.1). The nine children born with UCLP from the southern region had been treated with a one-stage palatal closure at a mean age of 13.6 months (range = 11–15 months). Five of these had previously undergone primary lip repair at a mean age of 4.5 months (range = 4–6 months), and four had undergone lip adhesion at a mean age of 3.75 months (range = 3–6 months) followed by primary lip repair at a mean age of 9 months (range = 6–11 months). In the UCLP group, eight children had received two or three sessions of speech therapy, four children 7–10 sessions, and one child 25 sessions. The remaining 16 children had received no speech therapy. Ethical approval The participation of the cleft centre in Gothenburg in the Scandcleft project was approved by the Regional Ethical Review Board of Gothenburg (R257–97). Enrol-
Hearing Hearing ability was assessed via pure tone audiometry (PTA) by a paediatric audiologist on the same day that the child’s speech and language abilities were assessed (table 1). The average PTA (measured at 500, 1000, 2000, 4000 Hz) of each ear was calculated. At 5 years of age, seven out of the 29 children in the UCLP group and three out of the 20 children in the COMP group were found to have mild hearing loss (21–40 dB HTL). The remaining children had normal hearing at the age of 5 years. At 3 years of age, 12 out of the 29 children in the UCLP group and two out of the 20 children in the COMP group had mild hearing loss. Five out of the seven children in the UCLP group and none out of the three children in the COMP group with hearing impairment at 5 years of age, also had a hearing impairment at 3 years of age. Hearing data was missing for two children in the UCLP group and one child in the COMP group. The remaining children had normal hearing. Recording procedure The documentation of the children from the western region at 5 years has previously been described in Klint¨o et al. (2011), and at 3 years of age in Klint¨o et al. (2014a). In summary, the children were audio and video recorded at 5 years of age (UCLP group: mean age = 60 months, range = 56–62 months; COMP group: mean age = 60 months, range = 54–64 months) and at 3 years of age (UCLP group: mean age = 36 months, range = 35–39 months; COMP group: mean age = 36 months, range = 35–38 months). In the group with UCLP treated with a two-stage closure with hard palate closure at about 12 months of age, one recording was missing and another child did not complete the session at 3 years. Speech was recorded with the cleft in the hard palate unrepaired in nine children with UCLP at 3 years of age. The recordings took place in a quiet room at Sahlgrenska University Hospital or at Sk˚ane University Hospital while interacting with a speech– language pathologist (SLP). The microphone was placed in front of the child slightly to the right and at a distance of 40 cm. For the children at Sahlgrenska University Hospital, speech was documented with digital audio recordings (Sony Walkman TCD-D8) using a condenser microphone (Sony ECM-MS957). In addition, simultaneous video recordings were made using a high quality
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Table 1. Number of children with hearing impairment (HI), 21–40 dB hearing threshold level TS12 (n = 11)
TS36 (n = 9)
OS (n = 9)
COMP (n = 20)
9 2 0
6 3 0
7 2 0
17 2 1
5 2 2 2
4 4 1 0
6 2 1 0
17 0 2 1
HI at 5 years No Unilateral Bilateral HI at 3 years No Unilateral Bilateral Missing
Note: TS12, two-stage repair with hard palate closure at 12 months of age; TS36, two-stage repair with hard palate closure at 36 months of age; OS, one-stage repair; COMP, comparison group without cleft lip and palate.
video camcorder with external microphone (Sony ECMMS957). Four audio recordings were of poor quality and were replaced by the audio files from the simultaneous video recordings. Audio files from video recordings were used for analysis of the children recorded at Sk˚ane University Hospital. The equipment used at Sk˚ane University Hospital was a video camera (Canon HF10) with an external microphone (Sony ECM-M5957). The BST (Renfrew 1997, Svensson and TuominenEriksson 2002) was used at 5 years of age. In addition, the children were presented with a single-word test by picture naming at 5 and 3 years of age. The test was developed in the Scandcleft Project to assess the production of consonants vulnerable to a cleft condition, i.e. oral stops and fricatives (Lohmander et al. 2009). It consisted of 33 pictures for eliciting single words at 5 years of age and 32 pictures (including two dummies) at 3 years of age. Thirty pictures/words were the same at both ages. An attempt by a child to produce the target word was counted as an elicited word, even if the production was unintelligible. If a child could not find the target word and semantic prompting failed, the SLP pronounced the target word and the child was asked to imitate. If the child refused to name the picture or used another word, the word was counted as not elicited. At 5 years of age, the median number of words elicited was 33 (range = 31–33) in both the UCLP and the COMP groups. The median number of words elicited with repetition at age 5 years was 4 (range = 1–8) in the UCLP group and 1 (range = 0–8) in the COMP group. The median number of words elicited at 3 years was 31 (range = 17–32) in the UCLP group and 31 (range = 25–32) in the COMP group. The median number of words elicited with repetition at 3 years of age was 11 (range 1–16) in the UCLP group and 8.5 (range 1–13) in the COMP group. The recordings were transferred to .wavfiles, edited in separate BST and word naming files. Assessment of the BST The BST files were prepared for blinded analysis, and orthographically transcribed and assessed according to the
test manual (Renfrew 1997, Svensson and TuominenEriksson 2002) by the study’s first author (main assessor) who has about nine years of experience working with children with cleft palate. The information score, MLU based on number of words, and the number of subordinate clauses was calculated for each child. After two months, nine recordings of the UCLP group and six recordings of the COMP group randomly chosen, were retranscribed and reassessed by the main assessor and an independent assessor. The latter is a SLP with about 6 years of experience working mainly with children with speech and language disorders not related to cleft palate. Before starting the assessment of the recordings, the two assessors transcribed and assessed recordings of BST of children with dyspraxia and/or phonological disorders and discussed the guidelines for assessment according to the manual. The information score reflects the amount information reproduced and that the content, context and sequence are correctly reproduced. Since the Swedish manual is vague regarding missing referents in a child’s narrative (Svensson and Tuominen-Eriksson 2002, Abrahamsson 2005), the guidelines of the English manual (Renfrew 1997) were followed instead. Consequently, score deduction was made only when the referent changed and the new referent was not obvious. Phonetic transcription and analysis of transcriptions In order to obtain a representative sample of the Swedish consonants, the whole words in the single-word test were transcribed narrowly, according to IPA and ExtIPA conventions (International Phonetic Association (IPA) 2002, 2005). Phonetic transcriptions completed by the first author from the audio recordings of the children documented at Sahlgrenska University Hospital at 5 (Klint¨o et al. 2011) and 3 years of age (Klint¨o et al. 2014a) were used. The first author also transcribed the audio files of the children documented at Sk˚ane University Hospital at the ages of 5 and 3 years, and three of the audio files, randomly chosen, were retranscribed one month later by the first author and the same second transcriber as in Klint¨o et al. (2011, 2014a).
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PCC-A at 5 and 3 years of age was calculated using the scoring rules for calculation of PCC described in Klint¨o et al. (2011), modified with respect to ageappropriate articulatory and phonological simplification processes. Phonemes established in 90% or more of children in the normative data (Bringfelt and Lindsta 2005, Klint¨o et al 2014a) were regarded as appropriate for those ages. Hence, substitutions or simplifications of phonemes used by more than 10% of children were accepted and scored as correct in PCC-A. Following these rules, varying types of lisp, such as inter-dental, lateral, supra-dental and palatal production of /s/, were accepted and scored as correct, along with weakening of /r/. Reliability testing Exact agreement was calculated by the intra-class correlation coefficient (ICC) with a two-way mixed effects model for the BST variables. Average ICC between the assessors was 0.969 for the information score, 0.973 for MLU and 0.863 for the number of subordinate clauses. Average ICC within the main assessor was 0.989 for the information score, 0.969 for MLU and 0.959 for the number of subordinate clauses. Based on retranscriptions of about 30% of the children (10 UCLP and seven COMP at age 5 years; nine UCLP and seven COMP at age 3 years), randomly chosen, inter- and intra-transcriber reliability of consonant transcriptions in the single-word test was calculated. The reliability was calculated by means of percentage agreement, point-by-point. The consonants compared had to be identically transcribed for place, manner and voicing in order to be considered as agreed. The four categories used for place were: bilabial/labiodental; inter-dental/dental/alveolar/postalveolar/retroflex (common variations of dental consonants in Swedish); palatal/velar/uvular (common variations of velars in co-articulation in Swedish); and glottal. The six categories used for manner were: stops; fricatives (lateral realization of /s/ included); nasals; realizations of /r/; the lateral /l/; and other approximants. Inter- and intra-transcriber agreement is presented in table 2. Statistical analysis Nonparametric statistics were used due to small group sizes and skewed distributions of data. The Mann– Whitney U-test was used for comparison of outcome measures between the UCLP and COMP groups at 5 years of age, and between girls and boys in the UCLP group. The Kruskal–Wallis test was used for comparisons among group medians for children with UCLP treated with different methods for primary palatal surgery. Correlations between the BST variables and PCC-A at 5 and 3 years of age were tested with
Spearman’s rank order correlation (confidence intervals given). Correlations between the BST variables and PTA levels for best and worst ear respectively were also tested with Spearman’s rank order correlation. For all statistical analyses, p < 0.05 (two-tailed) was considered to indicate significant differences. Results The median information score in the UCLP group was 14 (range = 0–31), the median MLU was 5.6 (range = 3.6–14.6), and median number of subordinate clauses 1 (range = 0–7) (table 3). No significant differences were found between the UCLP group and the COMP group regarding these measures. The statistical analysis indicated a strong trend towards a difference between groups regarding the information score (p = 0.051), with a higher score in the COMP group. However, the trend decreased (p = 0.065) when one child with attention deficit hyperactivity disorder was excluded from the UCLP group. Three children in the UCLP group and two in the COMP group, who declared that they were unwilling to participate, achieved low scores (UCLP: 0/3/8; COMP: 4/4). When they were excluded, no differences between the groups were found. The range of all three BST variables was wide in both groups. The mean norm value of the information score for Swedish 5-year-olds is 23 with a standard deviation (SD) of 6.21, according to the test manual (Svensson and TuominenEriksson 2002). The median information score of 14 in the UCLP group was more than 1 SD below this norm value. In the COMP group, the median score was 22, very close to the mean norm value and within 1 SD. Out of 29 children in the UCLP group, 19 (65.5%) had an information score below 1 SD of the norm value, compared with six out of 20 children (30%) in the COMP group. Furthermore, nine children in the UCLP group and two children in the COMP group scored 2 SD below the mean norm value. There were no significant differences among the three groups of children treated with different methods for primary palatal surgery (information score: x2 = 0.333, p = 0.847; MLU: x2 = 0.386, p = 0.825; number of subordinate clauses: x2 = 0.489, p = 0.783). Furthermore, there were no significant differences related to gender in the UCLP group (information score: z = –1.465, p = 0.143; MLU: z = –0.361, p = 0.718; number of subordinate clauses: z = –1.604, p = 0.109). Descriptive statistics on information score, and PCC-A at 3 and 5 years, categorized by surgical treatment (two children with missing data at age 3 years excluded), are presented in table 4. There were no significant correlations between the information score, MLU, and the number of subordinate clauses and PCC-A at 5 and 3 years of age in the
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Table 2. Percentage agreement of consonant transcriptions, point by point Median (minimum–maximum)
UCLP COMP UCLP + COMP
Inter 5 years
Intra 5 years
Inter 3 years
Intra 3 years
91 (47–97) 91 (84–99) 91 (47–99)
98 (91–99) 99 (95–100) 99 (91–100)
70 (62–80) 86 (79–93) 78 (62–93)
88 (79–92) 95 (93–99) 92 (79–99)
Note: Inter, inter-transcriber agreement; years = years of age; intra, intra-transcriber agreement; UCLP, group with unilateral cleft lip and palate; COMP, comparison group without cleft lip and palate.
Table 3. Comparison of Bus Story Test results between all children with unilateral cleft lip and palate (UCLP) and the comparison group without cleft palate (COMP) (Mann–Whitney U-test) UCLP, n = 29
COMP, n = 20
Outcome
Median (range)
Mean (SD)
Median (range)
Mean (SD)
Z
p
Information score Mean length of utterance (MLU) Number of subordinate clauses
14 (0–31) 5.6 (3.6–14.6) 1 (0–7)
16.4 (7.7) 6.2 (2.3) 1.8 (1.8)
22 (4–38) 7.5 (2.6–11.4) 1 (0–6)
21.4 (8.8) 7.3 (2.3) 1.9 (1.9)
–1.955 –1.823 –0.084
0.051 0.068 0.933
Note: SD, standard deviation.
Table 4. Descriptive statistics on information score, and percentage consonants adjusted for age (PCC-A) at 3 and 5 years, in the three groups of children treated with different methods for palatal surgery TS12 Outcome Information score PCC-A 5 years PCC-A 3 years
TS36
OS
Median (range)
Mean (SD)
Median (range)
Mean (SD)
Median (range)
Mean (SD)
14 (10–29) 94 (58–100) 77 (25–87)
17.0 (7.2) 87.9 (15.0) 62.7 (22.9)
17 (0–28) 95 (67–100) 37 (3–83)
16.2 (8.0) 90.8 (10.2) 43.1 (24.4)
21 (3–31) 100 (82–100) 84 (59–100)
17.3 (9.1) 97.5 (6.0) 81.4 (15.2)
Note: SD, standard deviation; TS, two-stage closure with hard palate closure at 12 months; TS36, two-stage closure with hard palate closure at 36 months; OS, one-stage closure.
Table 5. Spearman’s rho for the correlation analysis between Bus Story Test results (BST) at 5 years of age and percentage correct consonants adjusted for age (PCC-A) at 5 and 3 years of age in the group with unilateral cleft lip and palate (n = 29) PCC-A at 5 years
PCC-A at 3 years
BST at 5 years
rho
p
rho
p
Information score Mean length of utterance (MLU) Number of subordinate clauses
0.09 0.12 0.03
0.633 0.527 0.867
0.23 0.15 0.08
0.250 0.462 0.708
UCLP group (table 5). The relationship between hearing and the information score, MLU, and the number of subordinate clauses (best and worst PTA) at the age of 5 or 3 years in the UCLP group was also tested. No significant correlations were found (rho varied between –0.26 and 0.16). Discussion The purpose of this study was to investigate verbal competence in narrative retelling in 5-year-olds born with UCLP and to assess if verbal competence at the same age was related to articulation/phonology at 5 and at 3 years of age. Firstly, we assessed how 5-year-olds with UCLP performed on the information score, MLU, and the number of subordinate clauses when retelling, in com-
parison with children without cleft lip and palate. No significant differences were found. However, although no significant differences were found between the UCLP group and the COMP group, there was a strong trend towards poorer ability to retell information in the UCLP group. More than twice as large a proportion of the UCLP group than of the COMP group scored below 1 SD from the mean norm value. The median information score for the UCLP group was more than 1 SD below the norm, which is in accordance with findings in the unpublished study by Abrahamsson (2005). In that study, the group median for the information score in Swedish-speaking 7-year-olds with cleft palate was more than 1 SD below the norm. Similarly, Young et al. (2010) found that about one third of children with cleft palate scored below the 20th
126 percentile on a screening of grammar and vocabulary at about the same age. Thus, even if no significant differences in the expressive language variables between children with and without cleft palate on a group level have been found, a larger proportion of children with cleft palate seem to score lower on standardized tests of expressive language. Hence, these variables might differ significantly if the groups were larger. The mean and median values of MLU of both groups in the present study were close to the mean norm value of MLU of 5-year-olds ( = 6). Furthermore, the mean values of subordinate clauses of both groups in the present study were comparable to the mean norm value of 5year-olds ( = 2). The range was wide for both measures in both groups, which also is in congruence with the norm data. It has been stated that retelling generates longer and more complete stories and also more complex story grammar than freely produced narratives (Merrit and Liles 1989). However, about 10% of the children (three children in the UCLP group and two in the COMP group) declared that they did not want to retell the story. In these cases, the test-leader enticed the child to retell as much as possible, which resulted in information scores of 2 SD below the mean norm value. When these children were excluded from the statistical analysis no difference between the groups was found. There may be different reasons for a child not wanting to participate. Some may be related to linguistic performance, such as not understanding the task or difficulty remembering or reproducing the story. Others, such as shyness or not being in the mood to participate, may not. The test-leader’s conduct might in some cases have influenced the results of the BST. It is stated clearly in the manual that the test-leader should not ask leading questions since the response of the child in such cases cannot be used, but this occurred in a few instances in both groups. Another fact that may decrease the information score is when the referent is missing after changing the referent. Omission of the referent may not necessarily be due to linguistic difficulties. Since there is picture support and the child knows that the test-leader is familiar with the narrative, the referent may be implicit to the child. It is also possible that the child points out the referent in the picture. However, this information was unavailable since audio files were used for analysis. In a future study, it would be interesting to compare results on retelling in narratives with and without pictures visible to the examiner. Children with known additional malformations and/or syndromes were excluded from this study. However, other diagnoses, such as attention deficit hyperactivity disorder, may affect the results of the BST (Miniscalco et al. 2007). Thus, the group comparisons were recalculated when the child with diagnosed attention
Kristina Klint¨o et al. deficit hyperactivity disorder was excluded from the UCLP group. The trend towards a difference between groups then decreased somewhat. In studies on linguistic competence and cognition, children with cleft palate as a group often score below peers without cleft palate although within normal limits (e.g., Jocelyn et al. 1996, Broen et al. 1998). The explanation for this has yet to be clarified, but it could be that this is due to additional problems still not diagnosed. The findings highlight the necessity of collecting information on additional problems that may affect language development when assessing children born with cleft palate. To summarize, although there were no significant differences between the UCLP and COMP groups regarding the information score in narrative retelling, a noticeably larger proportion of the children with cleft palate had problems retelling information compared with their peers without UCLP and the group median was well below 1 SD of the norm. If larger groups of children had been compared, or if all children had participated willingly, a more significant difference might have appeared. Secondly, we wanted to assess possible differences among children with UCLP at the age of 5 years related to different methods for primary palatal surgery or gender, regarding the information score, MLU, and the number of subordinate clauses when retelling. No such differences were found. These findings may not be surprising since the groups were relatively small, and the method for primary palatal surgery is assumed primarily to affect speech outcome. In two studies, children with unoperated hard palate at age 3 years displayed more restricted phonology than peers with a closed hard palate (Willadsen 2012, Klint¨o et al. 2014b). Unfortunately, whether this difference persisted at age 5 or not was not studied. Thirdly, we wanted to assess if there was a relationship between the performance of the retelling task at 5 years of age and PCC-A at 5 and 3 years of age. True stop production (i.e. glides, pharyngeal, and glottal articulation excluded) in children with cleft palate, post surgery at 13 months, has been positively correlated to MLU and number of different words at 39 months of age (Chapman 2004). However, in the present study of older children, no correlations were found between PCC-A and the information score, MLU, and the number of subordinate clauses. Furthermore, no specific pattern was detected regarding the results of the BST and PCC-A when the children were studied individually. One example is the two children in the UCLP group with the highest information scores (31 and 29), of which one had a PCC-A score of 100% at 5 years of age while the other had a score of only 52%. In the UCLP group, 13 out of the 29 children had received therapy by an SLP between the ages of 3 and 5,
Verbal competence in 5-year-olds with cleft palate but just five of these children had received therapy for more than three sessions. No pattern regarding the relationship between speech and language difficulties and the number of SLP sessions was revealed when the children were reviewed individually. The number of therapy sessions a child attended seems not to have been related to displayed speech and language difficulties. The number of therapy sessions may be due to several causes including limited availability of resources, a low estimation of the individual child’s need for therapy, and the individual family’s lack of compliance with suggestions given by the local SLP. Since results of studies on toddlers with cleft palate have indicated that hearing may affect linguistic outcomes (Jocelyn et al. 1996, Broen et al. 1998), we also assessed the relationship between hearing and the BST variables. No significant correlations were found however. This was expected for several reasons. One is that OME and its associated hearing loss are fluctuating and therefore need to be assessed regularly and frequently in order to provide a reliable picture of the child’s hearing over the years. In the present study, only a few children presented hearing loss on the day of assessment. Furthermore, Young et al. (2010) found no correlations between history of OME and linguistic measures. In a study by Klint¨o et al. (2014a), the differences between Swedish-speaking children with and without UCLP on measures of articulation/phonology at 3 years of age persisted even after the possible effect of hearing loss was taken into account. This may be because the impact of hearing loss on linguistic ability in children with cleft palate is most prominent during the first years of life. Therefore, to what degree different language variables may be affected by hearing in the long term should be investigated further. Conclusion In this study, a larger proportion of the children with UCLP displayed problems retelling information compared with their peers without UCLP. There was a strong trend towards significant problems with retelling information in the UCLP group compared with the COMP group, which was not related to differences in the surgical method for primary palatal repair or to gender. Performance on the retelling task in the UCLP group was also not associated with articulatory/phonological competence, either at the same age or earlier. Since group size was small in both groups, further research is needed on narrative production in children with cleft palate. Acknowledgements The authors are grateful to SLP Maria Sporre who transcribed part of the material phonetically, to SLP Malin Schaar Johansson who assisted with assessment of the BST, to Dr Traci Flynn for sharing ˚ Nilsson for statistical expertise. the audiological data, and to Jan-Ake This research was supported by Stiftelsen Sunnerdahls handikapp-
127 fond, Region Sk˚ane and funds administered by Sk˚ane University Hospital. The study on which the paper was based has received ethical approval from the Research Ethics Committee of University of Gothenburg and University of Lund, and informed consent by the parents. The study was presented in part with a poster at the 15th International Meeting of the Clinical Phonetics and Linguistics Association, in Stockholm, Sweden, June 2014. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References ABRAHAMSSON, U., 2005, How do 7-year-olds with cleft lip and palate perform on the Bus Story Test: a study of language ability via a retelling task. MA thesis, Gothenburg University. [in Swedish] BISHOP, D. V. M. and EDMUNDSON, A., 1987, Language impaired 4-years-olds: distinguishing transient from persistent impairment. Journal of Speech and Hearing Disorders, 52, 156– 173. BOTTING, N., 2002, Narrative as a tool for the assessment of linguistic and pragmatic impairments. Child Language Teaching and Therapy, 18, 1–21. BRINGFELT, P.-A. and LINDSTA, K., 2005, Phonological analysis and normal data. In A. Lohmander, E. Borell, G. Henningsson, C. Havstam and C. Persson (eds.), SVANTE Svenskt artikulations- och nasalitets-test (Lund: Studentlitteratur), pp. 75–86. [in Swedish] BROEN, P. A., DEVERS, M. C., DOYLE, S. S., PROUTY, J. M. and MOLLER, K. T., 1998, Acquisition of linguistic and cognitive skills by children with cleft palate. Journal of Speech–Language and Hearing Research, 41, 676–687. CHAPMAN, K. L., 1993, Phonologic processes in children with cleft palate. Cleft Palate-Craniofacial Journal, 30, 64–72. CHAPMAN, K. L., 2004, Is presurgery and early postsurgery performance related to speech and language outcomes at 3 years of age for children with cleft palate? Clinical Linguistics and Phonetics, 18, 235–257. CHAPMAN, K. L., 2011, The relationship between early reading skills and speech and language performance in young children with cleft lip and palate. Cleft Palate-Craniofacial Journal, 48, 301– 311. COLLETT, B. R., LEROUX, B. and SPELTZ, M. L., 2010, Language and early reading among children with orofacial clefts. Cleft Palate-Craniofacial Journal, 47, 284–292. ¨ ¨ , C., JONSSON , R. and LOHMANDER, A., 2009, FLYNN, T., MOLLER The high prevalence of otitis media with effusion in children with cleft lip and palate as compared to children without clefts. International Journal of Pediatric Otorhinolaryngology, 73, 1441–1446. FREDERICKSON, M. S., CHAPMAN, K. L. and HARDIN-JONES, M., 2006, Conversational skills of children with cleft lip and palate: a reduplication and extension. Cleft Palate-Craniofacial Journal, 43, 179–188. FRIEDE, H., 2007, Maxillary growth controversies after two-stage palatal repair with delayed hard palate closure in unilateral cleft lip and palate patients: perspectives from literature and personal experience. Cleft Palate-Craniofacial Journal, 44, 129–136. INTERNATIONAL PHONETIC ASSOCIATION (IPA), 2002, 2005, The International Phonetic Alphabet (revised to 2005) (available at: http://www.langsci.ucl.ac.uk/ipa/IPA_chart _\050C\0512005.pdf); extIPA Symbols for Disordered Speech (revised to 2002) (available at: http://www.langsci .ucl.ac.uk/ipa/ExtIPAChart02.pdf) (accessed on 20 January 2014).
128 JOCELYN, L. J., PENKO, M. A. and RODE, H. L., 1996, Cognition, communication, and hearing in young children with cleft lip and palate and in control children: a longitudinal study. Pediatrics, 97, 529–534. KLINTO¨ , K., SALAMEH, E. K., SVENSSON, H. and LOHMANDER, A., 2011, The impact of speech material on speech judgement in children with and without cleft palate. International Journal of Language and Communication Disorders, 46, 348– 360. KLINTO¨ , K., SALAMEH, E. K., SVENSSON, H., OLSSON, M., FLYNN, T. and LOHMANDER, A., 2014a, Phonology in Swedish-speaking 3-year-olds born with cleft lip and palate and the relationship with consonant production at 18 months. International Journal of Language and Communication Disorders, 49, 240– 254. KLINTO¨ , K., SVENSSON, H., ELANDER, A. and LOHMANDER, A., 2014b, Speech and phonology in Swedish-speaking 3-yearolds with unilateral complete cleft lip and palate following different methods for primary palatal surgery. Cleft PalateCraniofacial Journal, 51, 274–282. KONST, E. M., RIETVELD, T., PETERS, H. F. and KUIJPERS-JAGTMAN, A. M., 2003, Language skills of young children with unilateral cleft lip and palate following infant orthopedics: a randomized clinical trial. Cleft Palate-Craniofacial Journal, 40, 356– 362. LEINONEN, E., LETTS, C. and SMITH, B. R., 2000, Narratives and storytelling. In E. Leinonen, C. Letts and B. R. Smith (eds), Children’s Pragmatic Communication Difficulties (London: Whurr), pp. 92–124. LOHMANDER, A., OLSSON, M. and FLYNN, T., 2011, Early consonant production in Swedish infants with and without unilateral cleft lip and palate and two-stage palatal repair. Cleft Palate-Craniofacial Journal, 48, 271– 285. LOHMANDER, A. and PERSSON, C., 2008, A longitudinal study of speech production in Swedish children with unilateral cleft lip and palate and two-stage palatal repair. Cleft PalateCraniofacial Journal, 45, 32–41. LOHMANDER, A., WILLADSEN, E., PERSSON, C., HENNINGSSON, G., BOWDEN, M. and HUTTERS, B., 2009, Methodology for speech assessment in the Scandcleft project—an international randomized clinical trial on palatal surgery: experiences from a pilot study. Cleft Palate-Craniofacial Journal, 46, 347– 362.
Kristina Klint¨o et al. MERRIT, D. D. and LILES, B. Z., 1989, Narrative analysis: clinical applications of story generation and story retelling. Journal of Speech and Hearing Disorders, 54, 438–447. MINISCALCO, C., HAGBERG, B., KADESJO¨ , B., WESTERLUND, M. and GILLBERG, C., 2007, Narrative skills, cognitive profiles and neuropsychiatric disorders in 7–8-year-old children with late developing language. International Journal of Language and Communication Disorders, 42, 665–681. MORRIS, H. and OZANNE, A., 2003, Phonetic, phonological, and language skills of children with a cleft palate. Cleft PalateCraniofacial Journal, 40, 460–470. PETERSON-FALZONE, S. J., 1996, The relationship between timing of cleft palate surgery and speech outcome: what have we learned, and where do we stand in the 1990s? Seminars in Orthodontics, 2, 185–191. RENFREW, C. E., 1997, Bus Story Test: A Test of Narrative Speech (Bicester: Winslow). SCHERER, N. J. and D’ANTONIO, L., 1995, Parent questionnaire for screening early language development in children with cleft palate. Cleft Palate-Craniofacial Journal, 32, 7–13. SEMB, G., 2001, Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate. Working paper presented at the WHO meeting, International Collaborative Research on Craniofacial Anomalies, Geneva, Switzerland, 5–8 November. SHRIBERG, L. D., 1993, Four new speech and prosody-voice measures for genetics research and other studies in developmental phonological disorders. Journal of Speech and Hearing Research, 36, 105–140. STOTHARD, S. E., SNOWLING, M. J., BISHOP, D. V. M., CHIPCASE, B. B. and KAPLAN, C. A., 1998, Language-impaired preschoolers. A follow-up into adolescence. Journal of Speech–Language and Hearing Research, 41, 407–418. SVENSSON, Y. and TUOMINEN-ERIKSSON, A. M., 2002, The Bus Story (Gothenburg: Specialpedagogiska institutet L¨aromedel). [in Swedish] WILLADSEN, E., 2012, Influence of timing of hard palate repair in a two-stage procedure on early language development in Danish children with cleft palate. Cleft Palate-Craniofacial Journal, 49, 574–595. YOUNG, S. E., PURCELL, A. A. and BALLARD, K. J., 2010, Expressive language skills in Chinese Singaporean preschoolers with nonsyndromic cleft lip and/or palate. International Journal of Pediatric Otorhinolaryngology, 74, 456–464.
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Research Report Verbal competence in narrative retelling in 5-year-olds with unilateral cleft lip and palate Kristina Klint¨o†‡, Eva-Kristina Salameh‡ and Anette Lohmander†§ †Division of Speech and Language Pathology, Karolinska Institutet, Stockholm, Sweden ‡Department of Otorhinolaryngology, Sk˚ane University Hospital, Malm¨o, Sweden §Department of Speech Pathology, Karolinska University Hospital, Stockholm, Sweden
(Received January 2014; accepted July 2014) Abstract Background: Research regarding expressive language performance in children born with cleft palate is sparse. The relationship between articulation/phonology and expressive language skills also needs to be further explored. Aims: To investigate verbal competence in narrative retelling in 5-year-old children born with unilateral cleft lip and palate (UCLP) and its possible relationship with articulation/phonology at 3 and 5 years of age. Methods & Procedures: A total of 49 children, 29 with UCLP treated according to three different procedures for primary palatal surgery and a comparison group of 20 children (COMP), were included. Longitudinally recorded audio files were used for analysis. At ages 3 and 5, the children were presented with a single-word test of word naming and at age 5 also the Bus Story Test (BST). The BST was assessed according to a test manual. The single-word test was phonetically transcribed and the percentage of consonants correct adjusted for age (PCC-A) was calculated. Differences regarding the BST results within the UCLP group were analysed. The results were compared with the results of the COMP group, and also with norm values. In addition, the relationship between the results of the BST and the PCC-A scores at ages 3 and 5 years was analysed. Outcomes & Results: No significant group differences or correlations were found. However, 65.5% of the children in the UCLP group had an information score below 1 standard deviation from the norm value compared with 30% in the COMP group. Conclusions: A larger proportion of children in the UCLP group than in the COMP group displayed problems with retelling but the differences between the two groups were not significant. There was no association between the BST results in the children with UCLP and previous or present articulatory/phonological competence. Since group size was small in both groups, the findings need to be verified in a larger study. Keywords: cleft palate, expressive language, narrative, articulation, phonology.
What this paper adds? What is already known on this subject? About half of the children born with cleft palate have difficulties with speech and phonology at about 3 years of age. Earlier studies point towards a relationship between early limitations of speech/phonology and general linguistic constraints in children born with cleft palate. What this study adds? This study investigated verbal competence in narrative retelling at 5 years of age in children with unilateral cleft lip and palate (UCLP) relative to peers without cleft palate and its relationship to articulation/phonology. A larger proportion of children with UCLP than without displayed problems in the retelling task, although the difference
Address correspondence to: Kristina Klint¨o, Karolinska Institutet, Department of Clinical Science, Intervention, and Technique, CLINTEC, Division of Speech and Language Pathology, B69, Karolinska University Hospital, Huddinge, S-141 86 Stockholm, Sweden; e-mail: [email protected] International Journal of Language & Communication Disorders C 2014 Royal College of Speech and Language Therapists ISSN 1368-2822 print/ISSN 1460-6984 online DOI: 10.1111/1460-6984.12127
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was not statistically significant. The performance on the retelling task was not related to previous or present articulatory/phonological competence.
Introduction Language development in children born with cleft palate has been sparsely explored. Even though the cleft is repaired at an early age, some children have persisting problems with speech and phonology the first years (e.g., Chapman 1993, Klint¨o et al. 2014a, b). Moreover, in studies on toddlers with cleft palate, the results have indicated limited expressive language ability compared with toddlers without cleft palate (e.g., Scherer and D’Antonio 1995, Jocelyn et al. 1996, Broen et al. 1998). Early limitations in speech and phonology in toddlers with cleft palate may be related to general linguistic constraints (e.g., Chapman 2004, Frederickson et al. 2006). Problems with speech and phonology may lead to reduced intelligibility in speech, which could have a negative impact on language development. The genesis of language difficulties in children with cleft palate thus needs to be clarified, to ensure that the affected children get the adequate support early in life. It is therefore useful to investigate expressive language in children with cleft palate further, as well as to consider its possible relationship to articulatory and phonological competence. The number of studies on expressive language in children with cleft palate compared with peers without cleft palate at pre-school or early school ages is limited and these studies show varying results (Konst et al. 2003, Collett et al. 2010, Young et al. 2010, Chapman 2011). For example, Collett et al. (2010) and Chapman (2011) found no significant differences between 5- and 6-year-olds with and without cleft palate regarding expressive vocabulary and grammatical skills. In a study of 12 Dutch-speaking 6-year-olds with UCLP, vocabulary and syntactic skills were within the normal range (Konst et al. 2003). In contrast, Young et al. (2010) demonstrated difficulties in the expressive use of grammar and vocabulary (below the 20th percentile on a standardized screening tool) in about one-third of 43 Chinese Singaporean children with cleft palate between 3;9 and 6;8 years of age. Significant differences in lexical ability and mean length of utterance (MLU) have been found at younger ages, i.e. between toddlers (16–30 months of age) with and without cleft palate (Scherer and D’Antonio 1995). Jocelyn et al. (1996) compared 16 children with clefts and 16 children without clefts who were matched for sex, ethnicity, socioeconomic status and birth order. The children with clefts scored significantly lower on expressive language at 12 and 24 months of age as well as
on formal tests of cognition and comprehension. Broen et al. (1998) found that children with cleft palate scored significantly lower on cognitive and linguistic measures than children without clefts at 24 months of age, although within the normal range. Percentage of consonants correct (PCC) is a measure of articulatory/phonological errors (Shriberg 1993). In several studies, children with cleft palate as a group displayed significantly lower PCC scores than children without cleft palate at both 5 (Lohmander and Persson 2008) and 3 years of age (Lohmander and Persson 2008, Willadsen 2012, Klint¨o et al. 2014a). However, other studies have accounted for individual differences that may explain these scores. For example, Morris and Ozanne (2003) identified a subgroup among children with cleft palate who had phonological language impairment. The degree of phonological problems seems to some extent to be influenced by surgical treatment. In two studies where the cleft in the soft palate was closed around 4–5 months of age, children with unoperated cleft in the hard palate at 3 years of age displayed more restricted phonology than children with a closed cleft in the hard palate at the same age (Willadsen 2012, Klint¨o et al. 2014b). Results from Chapman (1993) indicate that differences in phonological ability decrease with age between children with cleft palate as a group and children without cleft palate. In 5-year-olds with cleft palate, the total instances of phonological processes were similar to peers born without clefts. However, only 10 children at age 5 years were included in the study, and the results need to be confirmed through more studies. Frederickson et al. (2006) studied the relationship between speech production skills and conversational style in children with cleft palate between 33 and 44 months of age. Among them, 35% were characterized as either low in assertiveness or low in responsiveness. A significant positive correlation was seen between speech production skills and conversational assertiveness. The authors suggested several explanations for a passive communication style. These included the effects of true pragmatic deficit, poor intelligibility due to poor speech leading to unwillingness to speak, and shy personality. Children with a passive communication style may have fewer opportunities to practise their language, e.g. in narratives. Narrative production has been reported to be an ecologically valid task when assessing language skills in children (Botting 2002). Narrative ability may predict persistent language impairment (Bishop and Edmundson
Verbal competence in 5-year-olds with cleft palate 1987) and literacy performance (Stothard et al. 1998). To reproduce a narrative, several underlying abilities are needed, such as understanding of the task and the relationship between the input text and the topic, remembrance of the text, and also processing of the input text at the required speed (Leinonen et al. 2000). Pragmatic skills are also essential (Botting 2002). Retelling generates longer and more complete stories and more complex story grammar than freely produced narratives (Merrit and Liles 1989). The Bus Story Test (BST) is a standardized retelling test for assessment of verbal competence, where children are asked to retell a story with the aid of 12 pictures. The test may also indicate problems with phonology, semantic/lexical ability, grammar, sequencing, and language comprehension (Renfrew 1997). The BST has been translated into Swedish based on normative data from 100 Swedish-speaking children aged between 3;9 and 6;8 (Svensson and Tuominen-Eriksson 2002). In an unpublished study of 60 Swedish-speaking children at 7 years of age who were born with cleft palate, significant problems were displayed when reproducing information compared with the norm values of children born without cleft palate (Abrahamsson 2005). However, no results on verbal competence when retelling in children with cleft palate have been published to our knowledge. Method and technique for surgery is an important variable that could influence development of speech and phonology (Willadsen 2012, Klint¨o et al. 2014b). Different surgical methods are practiced. Some researchers argue that facial growth benefits from delayed palatal closure (Friede 2007), while speech development benefits from palatal closure as early as possible (PetersonFalzone 1996). Currently in Sweden, four cleft palate centres perform surgery in two stages, with soft palate closure at about 6 months of age and hard palate closure at about two years of age, in order to promote facial growth. The two other centres perform surgery in one stage at about 12 or 15 months of age. For speech and language development in children born with non-syndromic cleft palate, the single most important factor in addition to the cleft is hearing. It is well recognized that the incidence of otitis media with effusion (OME) and related mild to moderate hearing loss is high among children born with cleft palate (e.g., Flynn et al. 2009). Early hearing status has been correlated both with comprehension scores and expressive language at 24 months of age (Jocelyn et al. 1996) and with number of consonant types at 12 months (Lohmander et al. 2011). Broen et al. (1998) observed differences in cognitive and receptive language skills by 24 months in children with and without cleft palate, which, however, disappeared when hearing status and velopharyngeal function at 12 months were included as covariates. At the same time, no clear correlation between hearing status and language skill has been found in older children with cleft palate.
121 In a study by Young et al. (2010), a positive history of OME was not found to influence expressive language in 5- and 6-year-olds with cleft palate. However, only four of the 43 children had a hearing loss on the day of assessment, and the influence of hearing was not analysed. Thus, adequate knowledge regarding expressive language ability in children with cleft palate is lacking, as is understanding of the influence of hearing status. Expressive language difficulties at pre-school and early school age could have an impact on communication and learning during the school years, and need to be further explored. Aims The aim of the present study was therefore to investigate expressive language skills in terms of verbal competence in narrative retelling in children with UCLP at age 5 years and to compare these skills with present and previous articulatory/phonological ability. The specific research questions were:
r How do 5-year-olds with UCLP perform on the information score, MLU, and the number of subordinate clauses when retelling, compared with children without cleft palate? r Are there any differences among children with UCLP related to gender or different methods for primary palatal surgery, regarding the information score, MLU, and the number of subordinate clauses when retelling at the age of 5 years? In addition, we assessed the relationship between the information score, MLU, and the number of subordinate clauses when retelling at 5 years of age and the PCC adjusted for age (PCC-A) at the ages of 5 and 3 years in the children born with UCLP. Methods This is a prospective comparative study of 5-year-old children with UCLP and a comparison group, and a retrospective comparison at 3 years of age. Participants The participants included a consecutively selected group of 20 children born with UCLP between 1997 and 2003 from the western region of Sweden, a consecutively selected group of 10 children born with UCLP between 2005 and 2008 from the southern region of Sweden, and a comparison (COMP) group of 20 children born without cleft lip and palate in 2000. The children with UCLP from the western region of Sweden were participants in the Scandcleft project (Semb 2001). The children in the COMP group were recruited from child health centres
122
Kristina Klint¨o et al. ment of the patients in Malm¨o was approved by the Regional Ethical Review Board of Lund (D-nr: 548/2008). All parents had given written informed consent for participation.
49 children
11 two-stage closure (hp-closure ≈ 12 months)
29 UCLP (12 girls+17 boys)
20 COMP (11 girls+9 boys)
9 two-stage closure (hp-closure ≈ 36 months)
9 one-stage closure (≈ 13 months)
Figure 1. Number of children in the different groups. UCLP, children with unilateral cleft lip and palate; COMP, children without cleft lip and palate; hp-closure, hard palate closure.
in Gothenburg. According to a parental questionnaire, the COMP group was considered to be typically developing, including in terms of language development. No formal cognitive test was used. All children were monolingual Swedish-speaking and without any known additional malformations or syndromes. In the group with UCLP from the southern region, the recording of one child at age 5 years was missing. This resulted in 29 children (12 girls and 17 boys) in the UCLP group and 20 children (11 girls and nine boys) in the COMP group (figure 1). The 20 children in the UCLP group from the western region had been treated with early soft palate repair together with lip closure at a mean age of 4.6 months (range = 3.4–6.4 months). Eleven of these children had then been treated with hard palate closure at a mean age of 12.3 months (range = 11.6–13.7 months), and the remaining nine with hard palate closure at a mean age of 36.2 months (range = 35.6–37.1). The nine children born with UCLP from the southern region had been treated with a one-stage palatal closure at a mean age of 13.6 months (range = 11–15 months). Five of these had previously undergone primary lip repair at a mean age of 4.5 months (range = 4–6 months), and four had undergone lip adhesion at a mean age of 3.75 months (range = 3–6 months) followed by primary lip repair at a mean age of 9 months (range = 6–11 months). In the UCLP group, eight children had received two or three sessions of speech therapy, four children 7–10 sessions, and one child 25 sessions. The remaining 16 children had received no speech therapy. Ethical approval The participation of the cleft centre in Gothenburg in the Scandcleft project was approved by the Regional Ethical Review Board of Gothenburg (R257–97). Enrol-
Hearing Hearing ability was assessed via pure tone audiometry (PTA) by a paediatric audiologist on the same day that the child’s speech and language abilities were assessed (table 1). The average PTA (measured at 500, 1000, 2000, 4000 Hz) of each ear was calculated. At 5 years of age, seven out of the 29 children in the UCLP group and three out of the 20 children in the COMP group were found to have mild hearing loss (21–40 dB HTL). The remaining children had normal hearing at the age of 5 years. At 3 years of age, 12 out of the 29 children in the UCLP group and two out of the 20 children in the COMP group had mild hearing loss. Five out of the seven children in the UCLP group and none out of the three children in the COMP group with hearing impairment at 5 years of age, also had a hearing impairment at 3 years of age. Hearing data was missing for two children in the UCLP group and one child in the COMP group. The remaining children had normal hearing. Recording procedure The documentation of the children from the western region at 5 years has previously been described in Klint¨o et al. (2011), and at 3 years of age in Klint¨o et al. (2014a). In summary, the children were audio and video recorded at 5 years of age (UCLP group: mean age = 60 months, range = 56–62 months; COMP group: mean age = 60 months, range = 54–64 months) and at 3 years of age (UCLP group: mean age = 36 months, range = 35–39 months; COMP group: mean age = 36 months, range = 35–38 months). In the group with UCLP treated with a two-stage closure with hard palate closure at about 12 months of age, one recording was missing and another child did not complete the session at 3 years. Speech was recorded with the cleft in the hard palate unrepaired in nine children with UCLP at 3 years of age. The recordings took place in a quiet room at Sahlgrenska University Hospital or at Sk˚ane University Hospital while interacting with a speech– language pathologist (SLP). The microphone was placed in front of the child slightly to the right and at a distance of 40 cm. For the children at Sahlgrenska University Hospital, speech was documented with digital audio recordings (Sony Walkman TCD-D8) using a condenser microphone (Sony ECM-MS957). In addition, simultaneous video recordings were made using a high quality
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Table 1. Number of children with hearing impairment (HI), 21–40 dB hearing threshold level TS12 (n = 11)
TS36 (n = 9)
OS (n = 9)
COMP (n = 20)
9 2 0
6 3 0
7 2 0
17 2 1
5 2 2 2
4 4 1 0
6 2 1 0
17 0 2 1
HI at 5 years No Unilateral Bilateral HI at 3 years No Unilateral Bilateral Missing
Note: TS12, two-stage repair with hard palate closure at 12 months of age; TS36, two-stage repair with hard palate closure at 36 months of age; OS, one-stage repair; COMP, comparison group without cleft lip and palate.
video camcorder with external microphone (Sony ECMMS957). Four audio recordings were of poor quality and were replaced by the audio files from the simultaneous video recordings. Audio files from video recordings were used for analysis of the children recorded at Sk˚ane University Hospital. The equipment used at Sk˚ane University Hospital was a video camera (Canon HF10) with an external microphone (Sony ECM-M5957). The BST (Renfrew 1997, Svensson and TuominenEriksson 2002) was used at 5 years of age. In addition, the children were presented with a single-word test by picture naming at 5 and 3 years of age. The test was developed in the Scandcleft Project to assess the production of consonants vulnerable to a cleft condition, i.e. oral stops and fricatives (Lohmander et al. 2009). It consisted of 33 pictures for eliciting single words at 5 years of age and 32 pictures (including two dummies) at 3 years of age. Thirty pictures/words were the same at both ages. An attempt by a child to produce the target word was counted as an elicited word, even if the production was unintelligible. If a child could not find the target word and semantic prompting failed, the SLP pronounced the target word and the child was asked to imitate. If the child refused to name the picture or used another word, the word was counted as not elicited. At 5 years of age, the median number of words elicited was 33 (range = 31–33) in both the UCLP and the COMP groups. The median number of words elicited with repetition at age 5 years was 4 (range = 1–8) in the UCLP group and 1 (range = 0–8) in the COMP group. The median number of words elicited at 3 years was 31 (range = 17–32) in the UCLP group and 31 (range = 25–32) in the COMP group. The median number of words elicited with repetition at 3 years of age was 11 (range 1–16) in the UCLP group and 8.5 (range 1–13) in the COMP group. The recordings were transferred to .wavfiles, edited in separate BST and word naming files. Assessment of the BST The BST files were prepared for blinded analysis, and orthographically transcribed and assessed according to the
test manual (Renfrew 1997, Svensson and TuominenEriksson 2002) by the study’s first author (main assessor) who has about nine years of experience working with children with cleft palate. The information score, MLU based on number of words, and the number of subordinate clauses was calculated for each child. After two months, nine recordings of the UCLP group and six recordings of the COMP group randomly chosen, were retranscribed and reassessed by the main assessor and an independent assessor. The latter is a SLP with about 6 years of experience working mainly with children with speech and language disorders not related to cleft palate. Before starting the assessment of the recordings, the two assessors transcribed and assessed recordings of BST of children with dyspraxia and/or phonological disorders and discussed the guidelines for assessment according to the manual. The information score reflects the amount information reproduced and that the content, context and sequence are correctly reproduced. Since the Swedish manual is vague regarding missing referents in a child’s narrative (Svensson and Tuominen-Eriksson 2002, Abrahamsson 2005), the guidelines of the English manual (Renfrew 1997) were followed instead. Consequently, score deduction was made only when the referent changed and the new referent was not obvious. Phonetic transcription and analysis of transcriptions In order to obtain a representative sample of the Swedish consonants, the whole words in the single-word test were transcribed narrowly, according to IPA and ExtIPA conventions (International Phonetic Association (IPA) 2002, 2005). Phonetic transcriptions completed by the first author from the audio recordings of the children documented at Sahlgrenska University Hospital at 5 (Klint¨o et al. 2011) and 3 years of age (Klint¨o et al. 2014a) were used. The first author also transcribed the audio files of the children documented at Sk˚ane University Hospital at the ages of 5 and 3 years, and three of the audio files, randomly chosen, were retranscribed one month later by the first author and the same second transcriber as in Klint¨o et al. (2011, 2014a).
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PCC-A at 5 and 3 years of age was calculated using the scoring rules for calculation of PCC described in Klint¨o et al. (2011), modified with respect to ageappropriate articulatory and phonological simplification processes. Phonemes established in 90% or more of children in the normative data (Bringfelt and Lindsta 2005, Klint¨o et al 2014a) were regarded as appropriate for those ages. Hence, substitutions or simplifications of phonemes used by more than 10% of children were accepted and scored as correct in PCC-A. Following these rules, varying types of lisp, such as inter-dental, lateral, supra-dental and palatal production of /s/, were accepted and scored as correct, along with weakening of /r/. Reliability testing Exact agreement was calculated by the intra-class correlation coefficient (ICC) with a two-way mixed effects model for the BST variables. Average ICC between the assessors was 0.969 for the information score, 0.973 for MLU and 0.863 for the number of subordinate clauses. Average ICC within the main assessor was 0.989 for the information score, 0.969 for MLU and 0.959 for the number of subordinate clauses. Based on retranscriptions of about 30% of the children (10 UCLP and seven COMP at age 5 years; nine UCLP and seven COMP at age 3 years), randomly chosen, inter- and intra-transcriber reliability of consonant transcriptions in the single-word test was calculated. The reliability was calculated by means of percentage agreement, point-by-point. The consonants compared had to be identically transcribed for place, manner and voicing in order to be considered as agreed. The four categories used for place were: bilabial/labiodental; inter-dental/dental/alveolar/postalveolar/retroflex (common variations of dental consonants in Swedish); palatal/velar/uvular (common variations of velars in co-articulation in Swedish); and glottal. The six categories used for manner were: stops; fricatives (lateral realization of /s/ included); nasals; realizations of /r/; the lateral /l/; and other approximants. Inter- and intra-transcriber agreement is presented in table 2. Statistical analysis Nonparametric statistics were used due to small group sizes and skewed distributions of data. The Mann– Whitney U-test was used for comparison of outcome measures between the UCLP and COMP groups at 5 years of age, and between girls and boys in the UCLP group. The Kruskal–Wallis test was used for comparisons among group medians for children with UCLP treated with different methods for primary palatal surgery. Correlations between the BST variables and PCC-A at 5 and 3 years of age were tested with
Spearman’s rank order correlation (confidence intervals given). Correlations between the BST variables and PTA levels for best and worst ear respectively were also tested with Spearman’s rank order correlation. For all statistical analyses, p < 0.05 (two-tailed) was considered to indicate significant differences. Results The median information score in the UCLP group was 14 (range = 0–31), the median MLU was 5.6 (range = 3.6–14.6), and median number of subordinate clauses 1 (range = 0–7) (table 3). No significant differences were found between the UCLP group and the COMP group regarding these measures. The statistical analysis indicated a strong trend towards a difference between groups regarding the information score (p = 0.051), with a higher score in the COMP group. However, the trend decreased (p = 0.065) when one child with attention deficit hyperactivity disorder was excluded from the UCLP group. Three children in the UCLP group and two in the COMP group, who declared that they were unwilling to participate, achieved low scores (UCLP: 0/3/8; COMP: 4/4). When they were excluded, no differences between the groups were found. The range of all three BST variables was wide in both groups. The mean norm value of the information score for Swedish 5-year-olds is 23 with a standard deviation (SD) of 6.21, according to the test manual (Svensson and TuominenEriksson 2002). The median information score of 14 in the UCLP group was more than 1 SD below this norm value. In the COMP group, the median score was 22, very close to the mean norm value and within 1 SD. Out of 29 children in the UCLP group, 19 (65.5%) had an information score below 1 SD of the norm value, compared with six out of 20 children (30%) in the COMP group. Furthermore, nine children in the UCLP group and two children in the COMP group scored 2 SD below the mean norm value. There were no significant differences among the three groups of children treated with different methods for primary palatal surgery (information score: x2 = 0.333, p = 0.847; MLU: x2 = 0.386, p = 0.825; number of subordinate clauses: x2 = 0.489, p = 0.783). Furthermore, there were no significant differences related to gender in the UCLP group (information score: z = –1.465, p = 0.143; MLU: z = –0.361, p = 0.718; number of subordinate clauses: z = –1.604, p = 0.109). Descriptive statistics on information score, and PCC-A at 3 and 5 years, categorized by surgical treatment (two children with missing data at age 3 years excluded), are presented in table 4. There were no significant correlations between the information score, MLU, and the number of subordinate clauses and PCC-A at 5 and 3 years of age in the
Verbal competence in 5-year-olds with cleft palate
125
Table 2. Percentage agreement of consonant transcriptions, point by point Median (minimum–maximum)
UCLP COMP UCLP + COMP
Inter 5 years
Intra 5 years
Inter 3 years
Intra 3 years
91 (47–97) 91 (84–99) 91 (47–99)
98 (91–99) 99 (95–100) 99 (91–100)
70 (62–80) 86 (79–93) 78 (62–93)
88 (79–92) 95 (93–99) 92 (79–99)
Note: Inter, inter-transcriber agreement; years = years of age; intra, intra-transcriber agreement; UCLP, group with unilateral cleft lip and palate; COMP, comparison group without cleft lip and palate.
Table 3. Comparison of Bus Story Test results between all children with unilateral cleft lip and palate (UCLP) and the comparison group without cleft palate (COMP) (Mann–Whitney U-test) UCLP, n = 29
COMP, n = 20
Outcome
Median (range)
Mean (SD)
Median (range)
Mean (SD)
Z
p
Information score Mean length of utterance (MLU) Number of subordinate clauses
14 (0–31) 5.6 (3.6–14.6) 1 (0–7)
16.4 (7.7) 6.2 (2.3) 1.8 (1.8)
22 (4–38) 7.5 (2.6–11.4) 1 (0–6)
21.4 (8.8) 7.3 (2.3) 1.9 (1.9)
–1.955 –1.823 –0.084
0.051 0.068 0.933
Note: SD, standard deviation.
Table 4. Descriptive statistics on information score, and percentage consonants adjusted for age (PCC-A) at 3 and 5 years, in the three groups of children treated with different methods for palatal surgery TS12 Outcome Information score PCC-A 5 years PCC-A 3 years
TS36
OS
Median (range)
Mean (SD)
Median (range)
Mean (SD)
Median (range)
Mean (SD)
14 (10–29) 94 (58–100) 77 (25–87)
17.0 (7.2) 87.9 (15.0) 62.7 (22.9)
17 (0–28) 95 (67–100) 37 (3–83)
16.2 (8.0) 90.8 (10.2) 43.1 (24.4)
21 (3–31) 100 (82–100) 84 (59–100)
17.3 (9.1) 97.5 (6.0) 81.4 (15.2)
Note: SD, standard deviation; TS, two-stage closure with hard palate closure at 12 months; TS36, two-stage closure with hard palate closure at 36 months; OS, one-stage closure.
Table 5. Spearman’s rho for the correlation analysis between Bus Story Test results (BST) at 5 years of age and percentage correct consonants adjusted for age (PCC-A) at 5 and 3 years of age in the group with unilateral cleft lip and palate (n = 29) PCC-A at 5 years
PCC-A at 3 years
BST at 5 years
rho
p
rho
p
Information score Mean length of utterance (MLU) Number of subordinate clauses
0.09 0.12 0.03
0.633 0.527 0.867
0.23 0.15 0.08
0.250 0.462 0.708
UCLP group (table 5). The relationship between hearing and the information score, MLU, and the number of subordinate clauses (best and worst PTA) at the age of 5 or 3 years in the UCLP group was also tested. No significant correlations were found (rho varied between –0.26 and 0.16). Discussion The purpose of this study was to investigate verbal competence in narrative retelling in 5-year-olds born with UCLP and to assess if verbal competence at the same age was related to articulation/phonology at 5 and at 3 years of age. Firstly, we assessed how 5-year-olds with UCLP performed on the information score, MLU, and the number of subordinate clauses when retelling, in com-
parison with children without cleft lip and palate. No significant differences were found. However, although no significant differences were found between the UCLP group and the COMP group, there was a strong trend towards poorer ability to retell information in the UCLP group. More than twice as large a proportion of the UCLP group than of the COMP group scored below 1 SD from the mean norm value. The median information score for the UCLP group was more than 1 SD below the norm, which is in accordance with findings in the unpublished study by Abrahamsson (2005). In that study, the group median for the information score in Swedish-speaking 7-year-olds with cleft palate was more than 1 SD below the norm. Similarly, Young et al. (2010) found that about one third of children with cleft palate scored below the 20th
126 percentile on a screening of grammar and vocabulary at about the same age. Thus, even if no significant differences in the expressive language variables between children with and without cleft palate on a group level have been found, a larger proportion of children with cleft palate seem to score lower on standardized tests of expressive language. Hence, these variables might differ significantly if the groups were larger. The mean and median values of MLU of both groups in the present study were close to the mean norm value of MLU of 5-year-olds ( = 6). Furthermore, the mean values of subordinate clauses of both groups in the present study were comparable to the mean norm value of 5year-olds ( = 2). The range was wide for both measures in both groups, which also is in congruence with the norm data. It has been stated that retelling generates longer and more complete stories and also more complex story grammar than freely produced narratives (Merrit and Liles 1989). However, about 10% of the children (three children in the UCLP group and two in the COMP group) declared that they did not want to retell the story. In these cases, the test-leader enticed the child to retell as much as possible, which resulted in information scores of 2 SD below the mean norm value. When these children were excluded from the statistical analysis no difference between the groups was found. There may be different reasons for a child not wanting to participate. Some may be related to linguistic performance, such as not understanding the task or difficulty remembering or reproducing the story. Others, such as shyness or not being in the mood to participate, may not. The test-leader’s conduct might in some cases have influenced the results of the BST. It is stated clearly in the manual that the test-leader should not ask leading questions since the response of the child in such cases cannot be used, but this occurred in a few instances in both groups. Another fact that may decrease the information score is when the referent is missing after changing the referent. Omission of the referent may not necessarily be due to linguistic difficulties. Since there is picture support and the child knows that the test-leader is familiar with the narrative, the referent may be implicit to the child. It is also possible that the child points out the referent in the picture. However, this information was unavailable since audio files were used for analysis. In a future study, it would be interesting to compare results on retelling in narratives with and without pictures visible to the examiner. Children with known additional malformations and/or syndromes were excluded from this study. However, other diagnoses, such as attention deficit hyperactivity disorder, may affect the results of the BST (Miniscalco et al. 2007). Thus, the group comparisons were recalculated when the child with diagnosed attention
Kristina Klint¨o et al. deficit hyperactivity disorder was excluded from the UCLP group. The trend towards a difference between groups then decreased somewhat. In studies on linguistic competence and cognition, children with cleft palate as a group often score below peers without cleft palate although within normal limits (e.g., Jocelyn et al. 1996, Broen et al. 1998). The explanation for this has yet to be clarified, but it could be that this is due to additional problems still not diagnosed. The findings highlight the necessity of collecting information on additional problems that may affect language development when assessing children born with cleft palate. To summarize, although there were no significant differences between the UCLP and COMP groups regarding the information score in narrative retelling, a noticeably larger proportion of the children with cleft palate had problems retelling information compared with their peers without UCLP and the group median was well below 1 SD of the norm. If larger groups of children had been compared, or if all children had participated willingly, a more significant difference might have appeared. Secondly, we wanted to assess possible differences among children with UCLP at the age of 5 years related to different methods for primary palatal surgery or gender, regarding the information score, MLU, and the number of subordinate clauses when retelling. No such differences were found. These findings may not be surprising since the groups were relatively small, and the method for primary palatal surgery is assumed primarily to affect speech outcome. In two studies, children with unoperated hard palate at age 3 years displayed more restricted phonology than peers with a closed hard palate (Willadsen 2012, Klint¨o et al. 2014b). Unfortunately, whether this difference persisted at age 5 or not was not studied. Thirdly, we wanted to assess if there was a relationship between the performance of the retelling task at 5 years of age and PCC-A at 5 and 3 years of age. True stop production (i.e. glides, pharyngeal, and glottal articulation excluded) in children with cleft palate, post surgery at 13 months, has been positively correlated to MLU and number of different words at 39 months of age (Chapman 2004). However, in the present study of older children, no correlations were found between PCC-A and the information score, MLU, and the number of subordinate clauses. Furthermore, no specific pattern was detected regarding the results of the BST and PCC-A when the children were studied individually. One example is the two children in the UCLP group with the highest information scores (31 and 29), of which one had a PCC-A score of 100% at 5 years of age while the other had a score of only 52%. In the UCLP group, 13 out of the 29 children had received therapy by an SLP between the ages of 3 and 5,
Verbal competence in 5-year-olds with cleft palate but just five of these children had received therapy for more than three sessions. No pattern regarding the relationship between speech and language difficulties and the number of SLP sessions was revealed when the children were reviewed individually. The number of therapy sessions a child attended seems not to have been related to displayed speech and language difficulties. The number of therapy sessions may be due to several causes including limited availability of resources, a low estimation of the individual child’s need for therapy, and the individual family’s lack of compliance with suggestions given by the local SLP. Since results of studies on toddlers with cleft palate have indicated that hearing may affect linguistic outcomes (Jocelyn et al. 1996, Broen et al. 1998), we also assessed the relationship between hearing and the BST variables. No significant correlations were found however. This was expected for several reasons. One is that OME and its associated hearing loss are fluctuating and therefore need to be assessed regularly and frequently in order to provide a reliable picture of the child’s hearing over the years. In the present study, only a few children presented hearing loss on the day of assessment. Furthermore, Young et al. (2010) found no correlations between history of OME and linguistic measures. In a study by Klint¨o et al. (2014a), the differences between Swedish-speaking children with and without UCLP on measures of articulation/phonology at 3 years of age persisted even after the possible effect of hearing loss was taken into account. This may be because the impact of hearing loss on linguistic ability in children with cleft palate is most prominent during the first years of life. Therefore, to what degree different language variables may be affected by hearing in the long term should be investigated further. Conclusion In this study, a larger proportion of the children with UCLP displayed problems retelling information compared with their peers without UCLP. There was a strong trend towards significant problems with retelling information in the UCLP group compared with the COMP group, which was not related to differences in the surgical method for primary palatal repair or to gender. Performance on the retelling task in the UCLP group was also not associated with articulatory/phonological competence, either at the same age or earlier. Since group size was small in both groups, further research is needed on narrative production in children with cleft palate. Acknowledgements The authors are grateful to SLP Maria Sporre who transcribed part of the material phonetically, to SLP Malin Schaar Johansson who assisted with assessment of the BST, to Dr Traci Flynn for sharing ˚ Nilsson for statistical expertise. the audiological data, and to Jan-Ake This research was supported by Stiftelsen Sunnerdahls handikapp-
127 fond, Region Sk˚ane and funds administered by Sk˚ane University Hospital. The study on which the paper was based has received ethical approval from the Research Ethics Committee of University of Gothenburg and University of Lund, and informed consent by the parents. The study was presented in part with a poster at the 15th International Meeting of the Clinical Phonetics and Linguistics Association, in Stockholm, Sweden, June 2014. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
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